Genetic Information-Seeking Behaviors and Knowledge among Family Members and Patients with Inherited Bone Marrow Failure Syndromes

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• 作者：Jada G. Hamilton ; Sadie P. Hutson ; Amy E. Frohnmayer…
• 关键词：Genetic counseling ; Information seeking ; Genetic literacy ; Inherited bone marrow failure syndromes ; Fanconi anemia ; Dyskeratosis congenita ; Diamond ; Blackfan anemia ; Shwachman ; Diamond syndrome
• 刊名：Journal of Genetic Counseling
• 出版年：2015
• 出版时间：October 2015
• 年：2015
• 卷：24
• 期：5
• 页码：760-770
• 全文大小：294 KB
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• 作者单位：Jada G. Hamilton (1)
  Sadie P. Hutson (2)
  Amy E. Frohnmayer (3)
  Paul K. J. Han (4)
  June A. Peters (5)
  Ann G. Carr (6)
  Blanche P. Alter (5)
  
  1. Department of Psychiatry & Behavioral Sciences, Memorial Sloan Kettering Cancer Center, 641 Lexington Avenue, 7th floor, New York, NY, 10022, USA
  2. College of Nursing, University of Tennessee-Knoxville, Knoxville, TN, USA
  3. Clinical Mental Health Counseling Program, Oregon State University -Cascades, Bend, OR, USA
  4. Center for Outcomes Research and Evaluation, Maine Medical Center Research Institute, Scarborough, ME, USA
  5. Clinical Genetics Branch, Division of Cancer Epidemiology and Genetics, National Cancer Institute, Rockville, MD, USA
  6. Westat, Rockville, MD, USA
  
• 刊物类别：Biomedical and Life Sciences
• 刊物主题：Biomedicine
  Human Genetics
  Public Health
  Clinical Psychology
  Gynecology
  Ethics
  
• 出版者：Springer Netherlands
• ISSN：1573-3599

文摘

Inherited bone marrow failure syndromes (IBMFS) including Fanconi anemia, dyskeratosis congenita, Diamond-Blackfan anemia, and Shwachman-Diamond syndrome are rare genetic disorders characterized by hematologic complications and increased risk of cancer. Patients and their families likely experience obstacles in obtaining sufficient health information given their disorders-rarity. To investigate this possibility, we examined information-seeking behaviors and levels of general and disorder-specific genetic knowledge among 315 members of 174 families with an IBMFS, and how information-seeking behaviors and socio-demographic factors may be associated with their genetic knowledge. Cross-sectional survey data indicated that participants were most likely to have ever used the Internet or healthcare providers for genetic information. On average, participants correctly answered 57 % of items assessing general genetic knowledge and 49-9 % of disorder-specific knowledge items. Greater knowledge was associated with greater education and ever experiencing genetic counseling, attending a scientific meeting, and seeking information from the Internet and scientific literature. Among families with Fanconi anemia (whose family support organization has the longest history of providing information), greater disorder-specific genetic knowledge was also associated with seeking information from support groups and other affected families. Results suggest that families with IBMFS have uncertainty regarding genetic aspects of their disorder, and highlight potential channels for delivering educational resources. Keywords Genetic counseling Information seeking Genetic literacy Inherited bone marrow failure syndromes Fanconi anemia Dyskeratosis congenita Diamond-Blackfan anemia Shwachman-Diamond syndrome